Rituximab induces resolution of recurrent diffuse alveolar hemorrhage in a patient with primary antiphospholipid antibody syndrome

Diffuse alveolar hemorrhage (DAH) is a rare manifestation of primary antiphospholipid antibody syndrome (APS). We describe a patient with primary APS and refractory recurrent episodes of DAH. The patient was admitted 15 times due to recurrent episodes of DAH in a period of 18 months. Multiple immuno...

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Bibliographic Details
Published in:	Lupus Vol. 21; no. 4; pp. 438 - 440
Main Authors:	Elazary, A Scheiman, Klahr, PP, Hershko, AY, Dranitzki, Z, Rubinow, A, Naparstek, Y
Format:	Journal Article
Language:	English
Published:	London, England SAGE Publications 01-04-2012 Sage Publications Ltd
Subjects:	Adult Alveoli Antibodies Antibodies, Monoclonal, Murine-Derived - therapeutic use antiphospholipid antibodies Antiphospholipid Syndrome - blood Antiphospholipid Syndrome - complications Antiphospholipid Syndrome - drug therapy Antiphospholipid Syndrome - immunology Autoantibodies - blood Biomarkers - blood cardiolipin Case reports Drug dosages Glycoproteins Hemoptysis Hemorrhage Hemorrhage - drug therapy Hemorrhage - etiology Humans Immunoglobulin G Immunologic Factors - therapeutic use Immunosuppressive agents Lung Diseases - drug therapy Lung Diseases - etiology Lupus Lymphoma Male Medicine Recurrence Rituximab Steroids Time Factors Treatment Outcome Jerusalem Israel Israel alveolar diffuse antiphospholipid rituximab hemorrhage
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Summary:	Diffuse alveolar hemorrhage (DAH) is a rare manifestation of primary antiphospholipid antibody syndrome (APS). We describe a patient with primary APS and refractory recurrent episodes of DAH. The patient was admitted 15 times due to recurrent episodes of DAH in a period of 18 months. Multiple immunosuppressive drugs did not improve his condition. Two years after his presentation, he was treated with rituximab (two doses of 1 g, 2 weeks apart). Six months later, the attacks of DAH have gradually disappeared. In a follow-up of more than 2 years after he received rituximab, the patient has had no further admissions due to DAH. Levels of antiphospholipid antibodies were measured during follow-up of 4 years. Anti-β2 glycoprotein IgG titer decreased to normal 6 months after therapy but anticardiolipin (aCL) antibody titer increased. We conclude that rituximab caused a dramatic clinical response in this patient. Anti-β2 glycoprotein IgG correlated better with the clinical response in this patient than aCL.
Bibliography:	ObjectType-Article-2 SourceType-Scholarly Journals-1 ObjectType-Feature-1 content type line 23 ObjectType-Case Study-2 ObjectType-Feature-4 ObjectType-Report-1 ObjectType-Article-3
ISSN:	0961-2033 1477-0962
DOI:	10.1177/0961203311422713